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Choking in the Night Due to NFLE Seizures in a Patient with Comorbid OSA

2014; American Academy of Sleep Medicine; Volume: 10; Issue: 10 Linguagem: Inglês

10.5664/jcsm.4122

1550-9397

Michele Terzaghi, Ivana Sartori, Riccardo Cremascoli, Valter Rustioni, Raffaele Manni,

Restraint-Related Deaths

Free AccessCPAPChoking in the Night Due to NFLE Seizures in a Patient with Comorbid OSA Michele Terzaghi, M.D., Ivana Sartori, M.D., Riccardo Cremascoli, M.D., Valter Rustioni, M.D., Raffaele Manni, M.D. Michele Terzaghi, M.D. Address correspondence to: Michele Terzaghi, M.D., Sleep Medicine and Epilepsy Unit, IRCCS National Neurological Institute C. Mondino Foundation, Via Mondino 2, 27100 Pavia, Italy+390382380316+390382380286 E-mail Address: [email protected] Sleep Medicine and Epilepsy Unit - C. Mondino National Neurological Institute, Pavia, Italy , Ivana Sartori, M.D. Epilepsy Surgery Centre "C. Munari" Sleep Disorders Centre, Niguarda Hospital, Milan, Italy , Riccardo Cremascoli, M.D. Sleep Medicine and Epilepsy Unit - C. Mondino National Neurological Institute, Pavia, Italy , Valter Rustioni, M.D. Sleep Medicine and Epilepsy Unit - C. Mondino National Neurological Institute, Pavia, Italy , Raffaele Manni, M.D. Sleep Medicine and Epilepsy Unit - C. Mondino National Neurological Institute, Pavia, Italy Published Online:October 15, 2014https://doi.org/10.5664/jcsm.4122Cited by:4SectionsAbstractPDFSupplemental Material ShareShare onFacebookTwitterLinkedInRedditEmail ToolsAdd to favoritesDownload CitationsTrack Citations AboutABSTRACTAwakenings from sleep with gasping and feeling of choking can be due to nocturnal frontal lobe epilepsy (NFLE) as well as sleep apnea (OSA). We describe the case of an overweight man, referred to us with suspected OSA and reporting awakenings from sleep accompanied by gasping and a choking feeling, which proved to be, after investigation, NFLE seizures in a patient with comorbid OSA. We underline that gasping or choking on awakening, especially when accompanied by abnormal motor-behavioral manifestations, should be interpreted with caution. Careful investigation by means of video-polysomnography is warranted in selected cases, including patients with a strong clinical suspicion of sleep apnea.Citation:Terzaghi M, Sartori I, Cremascoli R, Rustioni V, Manni R. Choking in the night due to NFLE seizures in a patient with comorbid OSA. J Clin Sleep Med 2014;10(10):1149-1152.INTRODUCTIONAwakenings accompanied by gasping and a feeling of choking associated with abnormal motor activity can be interpreted as paroxysmal arousals of the kind manifested in nocturnal frontal lobe epilepsy (NFLE) and in sleep apnea (OSA). The differential diagnosis between NFLE and OSA can be challenging, and overlapping features can result in NFLE being misdiagnosed as OSA.1,2 In particular, sudden awakening with a choking feeling and excessive daytime sleepiness have been found to be overlapping features of NFLE and OSA.2We describe the case of a patient who was referred to us with clinical suspicion of OSA based on reported episodes of gasping and a choking feeling on awakening from sleep.This patient was ultimately diagnosed with NFLE and co-morbid OSA.REPORT OF CASEA 33-year-old man was referred to our sleep medicine outpatient unit with clinical suspicion of OSA, as he reported a history of snoring and, since a few months, nocturnal episodes of gasping and choking, sometimes with a sore and burning throat and an urge to clear the throat. The occurrence of snoring and apneas during sleep was confirmed by the patient's wife, who also reported that the patient would sometimes abruptly sit up in bed, gasping for air, crying out and looking around for help.These events, lasting just a few seconds, occurred 4-5 times per night almost every night (the patient reported a maximum of one seizure-free night per week). They were not accompanied by abnormal motor manifestations (such as tonic/dystonic posturing, involuntary movements, clonic jerks, pelvic thrusting); consciousness was preserved throughout seizures and, when the episodes ended, the patient was always able to remember and describe them clearly. No postictal confusion was reported.The patient's BMI was 29.7 kg/m2; no excessive daytime sleepiness was reported (Epworth Sleepiness Scale score 8). The patient's personal and family history was negative for epilepsy and arousal parasomnias. Neurological examination, routine EEG, esophageal pH manometry, and brain MRI were normal. Oropharyngeal examination revealed a mild deviation of the nasal septum, a long uvula, and Mallampati grade I presentation of the soft palate; Muller's maneuver showed about 50% narrowing of the velopharyngeal opening.Video-PSG AnalysisBaselineThe patient, having given his informed consent, underwent in-lab video-polysomnography (video-PSG) with full-scalp EEG and standard respiratory monitoring including use of a microphone and abdominal and thoracic strain gauges; airflow was recorded by means of thermocouples located at the mouth and nostrils, and SpO2 using a fingertip pulse oximeter. Two stereotyped episodes occurring on abrupt arousals from slow wave sleep were recorded: the patient suddenly sat up in bed repeatedly clearing his throat and crying out for help. When questioned by the technician, he appeared to be lucid and reported gasping and choking sensations; however, although consciousness was preserved during the events, the patient was not able to accurately describe what happened in the immediate postictal state: in fact, he reports a sensation of choking, but does not remember his rather dramatic screaming (Videos 1 and 2).The video-PSG recording (Figure 1, Video 1) showed the presence of snoring without any clear-cut respiratory abnormalities while the EEG before, during and after the episode did not show any epileptiform or slow activities. Respiratory analysis showed snoring during NREM and REM sleep and obstructive apneas and hypopneas exclusively during REM sleep, with a global apnea-hypopnea index (AHI) and an oxygen desaturation index of 8/h of sleep (corresponding to a REM AHI and REM oxygen desaturation index of 79.2/h).Figure 1 Upper part: Paroxysmal arousal occurring during stage 3 NREM sleep. Note the recurrence of snoring in the presence of an otherwise regular respiratory pattern. Bottom part: hypnogram and respiratory pattern throughout the recorded night. The black arrow indicates the occurrence of the paroxysmal arousal. EOG1+ EOG1-, electroculogram; MILO+ MILO-, chin EMG; ECG1+ ECG1-, electrocardiogram; THOR+ THOR-, thoracic respiratory movements; ABDM+ ABDM-, abdominal respiratory movements; FLOW- FLOW+, oro-nasal airflow; MIC+ MIC-, snoring; TibR+ TibR-, right anterior tibialis EMG; TibL+ TibL-, left anterior tibialis EMG; SpO2- SpO2+, arterial oxygen hemoglobin saturation; BODY+ BODY-, body position.Download FigureIn the light of the clinical and video-PSG findings, the episodes captured (which the patient recognized as the same as those that had prompted him to seek medical advice) were interpreted as paroxysmal arousals within the semiological spectrum of NFLE. However, since one of the two episodes occurred while the patient was snoring, it was deemed necessary to rule out any potential role of sleep disordered breathing in triggering the paroxysmal events. Therefore, a CPAP titration was performed and video-PSG was repeated with the patient under CPAP treatment.CPAP TreatmentVideo-PSG during CPAP (7 cm H2O) showed persistence of the paroxysmal episodes (Figure 2, Video 2) in the absence of snoring and sleep disordered breathing events. The episodes showed the same features as those recorded during video-PSG without CPAP. The paroxysmal manifestations remitted when the patient was put on carbamazepine 800 mg/day (single bedtime dose).Figure 2 Upper part: Paroxysmal arousal occurring during stage 3 NREM sleep while the subject was under CPAP treatment; note the presence of a regular respiratory pattern. Bottom part: hypnogram and respiratory pattern throughout the recorded night. Black arrows indicate the occurrence of paroxysmal arousals. EOG1+ EOG1-, electroculogram; MILO+ MILO-, chin EMG; ECG1+ ECG1-, electrocardiogram; THOR+ THOR-, thoracic respiratory movements; ABDM+ ABDM-, abdominal respiratory movements; FLOW- FLOW+, oro-nasal airflow; MIC+ MIC-, snoring; TibR+ TibR-, right anterior tibialis EMG; TibL+ TibL-, left anterior tibialis EMG; SpO2- SpO2+, arterial oxygen hemoglobin saturation; BODY+ BODY-, body position.Download FigureDISCUSSIONMisdiagnosis of NFLE as sleep apnea syndrome has previously been reported in adult patients reporting a choking feeling on awakening, abnormal motor activity during sleep, and excessive daytime sleepiness.2 Choking at night as a major manifestation of NFLE has recently been reported in a child.3In the case here described, the diagnosis was particularly difficult since the clinical presentation consisted mainly of respiratory symptoms in the ictal phase, along with snoring and apneas (witnessed by the bed partner) in an overweight man. Imperfect recollection by the patient (i.e., omission of motor-behavioral events) could easily lead to misdiagnosis, especially without clarification of a bed partner.Objective analysis and characterization of nocturnal events captured by video-PSG is warranted in challenging cases. Indeed, it allowed us to diagnose the reported ictal nocturnal episodes of breathlessness as NFLE seizures in a patient with NFLE and comorbid OSA.Furthermore, the diagnosis of NFLE is often challenging because, as in this case, ictal scalp EEG is frequently normal.4 The evolution of epileptiform discharges either in deep brain structures or structures tangential to recording scalp electrodes is deemed to account for the lack of scalp EEG abnormalities; indeed, viscerosensitive symptoms like laryngeal and throat disturbances and breathing discomfort have been reported to characterize an insular origin of seizures and to arise in response to electrical stimulation of the anterior part of the insula.5,6 Extra-insular spreading of epileptic discharge to frontomesial regions is instead deemed to entrain complex motor manifestations.5DISCLOSURE STATEMENTThis was not an industry supported study. The authors have indicated no financial conflicts of interest.REFERENCES1 Manni R, Terzaghi MComorbidity between epilepsy and sleep disorders. Epilepsy Res; 2010;90:171-7, 20570109. CrossrefGoogle Scholar2 Oldani A, Zucconi M, Castronovo C, Ferini-Strambi LNocturnal frontal lobe epilepsy misdiagnosed as sleep apnea syndrome. Acta Neurol Scand; 1998;98:67-71, 9696531. CrossrefGoogle Scholar3 Rathore G, Larsen P, Parakh M, Fernandez CChoking at night: a case of opercular nocturnal frontal lobe epilepsy. Case Rep Pediatr; 2013;2013:606385, 24383033. Google Scholar4 Provini F, Plazzi G, Tinuper P, Vandi S, Lugaresi E, Montagna PNocturnal frontal lobe epilepsy. A clinical and polygraphic overview of 100 consecutive cases. Brain; 1999;122:1017-31, 10356056. CrossrefGoogle Scholar5 Proserpio P, Cossu M, Francione Set al.Insular-opercular seizures manifesting with sleep-related paroxysmal motor behaviors: a stereo-EEG study. Epilepsia; 2011;52:1781-91, 21883183. CrossrefGoogle Scholar6 Isnard J, Guenot M, Sindou M, Mauguire FClinical manifestations of insular lobe seizures: a stereo-electroencephalographic study. Epilepsia; 2004;45:1079-90, 15329073. CrossrefGoogle Scholar Previous article Next article FiguresReferencesRelatedDetailsCited by Diagnosis and treatment of epilepsy and sleep apnea comorbidityLiu F and Wang X Expert Review of Neurotherapeutics, 10.1080/14737175.2017.1262259, Vol. 17, No. 5, (475-485), Online publication date: 4-May-2017. Frontal Lobe Epilepsy: A Primer for Psychiatrists and a Systematic Review of Psychiatric ManifestationsGold J, Sher Y and Maldonado J Psychosomatics, 10.1016/j.psym.2016.05.005, Vol. 57, No. 5, (445-464), Online publication date: 1-Sep-2016. Obstructive sleep apnea in drug-resistant epilepsy: A significant comorbidity warranting diagnosis and treatmentDinkelacker V Revue Neurologique, 10.1016/j.neurol.2016.03.007, Vol. 172, No. 6-7, (361-370), Online publication date: 1-Jun-2016. Association Between Benzodiazepine Use and Epilepsy OccurrenceHarnod T, Wang Y and Kao C Medicine, 10.1097/MD.0000000000001571, Vol. 94, No. 37, (e1571), Online publication date: 1-Sep-2015. Volume 10 • Issue 10 • October 15, 2014ISSN (print): 1550-9389ISSN (online): 1550-9397Frequency: Monthly Metrics History Submitted for publicationMarch 1, 2014Submitted in final revised formJune 1, 2014Accepted for publicationJune 1, 2014Published onlineOctober 15, 2014 Information© 2014 American Academy of Sleep MedicineKeywordsepilepsysleep apneanocturnal frontal lobe epilepsysleepPDF download